Unless otherwise noted, the content of this course material is licensed under a Creative Commons Attribution - Non-Commercial - Share Alike 3.0 License.

Copyright 2008, Arno Kumagai, Gary Hammer

The following information is intended to inform and educate and is not a tool for self-diagnosis or a replacement for medical evaluation, advice, diagnosis or treatment by a healthcare professional. You should speak to your physician or make an appointment to be seen if you have questions or concerns about this information or your medical condition. You assume all responsibility for use and potential liability associated with any use of the material.

Material contains copyrighted content, used in accordance with U.S. law. Copyright holders of content included in this material should contact [email protected] with any questions, corrections, or clarifications regarding the use of content. The Regents of the University of Michigan do not license the use of third party content posted to this site unless such a license is specifically granted in connection with particular content objects. Users of content are responsible for their compliance with applicable law. Adrenal Physiology & Steroid Pharmacology

Logo: All Rights Reserved Regents of the University of Michigan.

2008 Gary D. Hammer, M.D., Ph.D. University of Michigan Ann Arbor, Michigan USA Learning Objectives

After this lecture you should have an understanding of:

• The feedback loops regulating secretion.

• The physiologic actions of glucocorticoids (cortisol) + mineralocorticoids ()

• The major pharmacologic uses of glucocorticoids.

• The major types of glucocorticoids.

• The major side effects of glucocorticoid therapy. Anatomy of the adrenal glands

Public Domain Wikimedia Commons

Public Domain Seers.gov

Public Domain Wikimedia Commons Histology of the

adrenal cortex

CC:BY-NC-SA 1.0 BY: Royal College of Surgeons of Ireland (RCSI)

adrenal medulla

Public Domain Wikimedia Commons Adrenocortical Hormones = Steroids

GLUCOCORTICOID MINERALOCORTICOID Cortisol Aldosterone

cortex medulla

Adrenal Steroidogenesis

glomerulosa

fasciculata

reticularis

gonad periphery

Public Domain Wikimedia Commons ‘Roids: The Bottom Line

In the right amounts, steroids can be the body’s best friend….

or in the wrong amounts, the body’s worst enemy…. Role of Glucocorticoids in Human Physiology

In the right amounts, glucocorticoids keep:

• Your blood pressure up (maintain cardiovascular stability).

• Your blood sugar up (maintain metabolic homeostasis).

• Your disposition sunny (maintain integrity of CNS function).

• Your temperament cool (regulate response to stress). Adrenocortical Hormones = Steroids

GLUCOCORTICOID MINERALOCORTICOID Cortisol Aldosterone

cortex medulla

Adrenal The HPA axis

Neural Stimuli Hypothalamus -- CRF ++ ACTH Anterior Pituitary --

ACTH Plasma Cortisol Concentration

Adrenal ++

CC:BY 3.0 BY: Regents of the University of Michigan Regulation of ACTH Expression by CRH

Public Domain Bruno Dubuc Post-translational Processing of POMC in the Normal Pituitary

POMC = Pro-opiomelanocortin

ACTH

Source: Undetermined MSH = Melanocyte stimulating hormone ACTH and Steroid Biosynthesis

POLYSOME CELL MEMBRANE ATP 3',5' cAMP INACTIVE PROTEIN PROTEIN ACTH ADENYLATE CYCLASE KINASE

ACTIVE PROTEIN

CHOLESTEROL ESTER  STEROID CHOLESTEROL BIOSYNTHESIS Secretion, Transport and Metabolism of Cortisol

Image of CNS Control removed Circadian Rhythm of Cortisol Secretion

Highest in morning

24 hour period 24 hour period day night day night 20 g/100 ml

μ 15

10

5 Plasma Cortisol Cortisol Plasma

0 8 AM 4 PM 12 MID 8 am 4 PM 12 MID

Source: Undetermined Lowest in evening Corticosteroid Binding Globulin (CBG)

Image of Globulin removed

SHBG Grishkovskaya et al, 1999

• Acidic glycoprotein MW 52,000 • Produced in liver, lung, kidney, testes • Regulates delivery of cortisol to tissues Conditions that Affect Cortisol Metabolism

• Increased Turnover: --Thyroxine --Barbiturates --Phenytoin

• Decreased Turnover: --Liver disease

• Increased Binding: --Estrogens Molecular Action of Glucocorticoids

Glucocorticoid receptors (GR) are transcriptional activators of a variety of gene products.

Image of Glucocorticoid Target Cell removed Metabolic Effects of Glucocorticoids

Prototypical Glucocorticoid = Cortisol

Glucocorticoids  Insulin

Glucocorticoids effects are generally opposite those of insulin. Glucocorticoids & Carbohydrate Metabolism

GLUCOCORTICOIDS Glucocorticoids increase hepatic (+) Glucocorticoids glucose output decrease insulin sensitivity (-) Liver Glucose (-) MUSCLE

(+) INSULIN

FAT CELL Glucocorticoid Effects on Protein Metabolism

Insulin Glucocorticoids

Anabolism (storage) Catabolism MUSCLE Protein synthesis Protein synthesis Protein breakdown Protein breakdown Amino acid release Amino acid release Glucocorticoid Effects on Lipid Metabolism

Insulin Glucocorticoids

Anabolism (storage) Catabolism Lipid synthesis Lipid synthesis Lipolysis ADIPOCYTE Lipolysis Fatty acid release Fatty acid release

Redistribution of fat Redistribution of Fat in Glucocorticoid Excess

Image of patient removed

Central obesity seen in Cushing’s Syndrome (Glucocorticoid Excess) Glucocorticoid Effects on Inflammatory Mediators

Glucocorticoids INHIBIT inflammation.

Inhibit:

1) Arachidonic acid and its metabolites (prostaglandins; leukotrienes) 2) Platelet activating factor (PAF) 3) Tumor necrosis factor (TNF) 4) Interleukin-1 (IL-1) 5) Plasminogen activator Sites of Action of Glucocorticoids in the Responses of Leukocytes During Antigenic Challenge/Inflammation

Image of Glucocorticoids removed Glucocorticoids

Clinical Uses of Glucocorticoids Steroid Therapy: Routes of Administration

• Systemic Oral Parenteral

• Topical • Inhalation Clinical Uses of Glucocorticoids

• Replacement therapy

• Anti-inflammatory effect

• Immunosuppression

• Androgen suppression Glucocorticoids: Use as Anti-Inflammatory Agents

Severe RA of hands Source: Undetermined

Emily Janz, a 36-year old woman presents with a 3-year history of rheumatoid arthritis. The disease has been progressive with involvement of PIP joints in both hands, wrists, elbows and TM joints. Treatment with non- steroidal anti-inflammatory drugs (NSAIDs) has not been successful.

Treatment with prednisone is begun using an alternate-day program. Glucocorticoids: Use in Immunosuppression

CC-BY-ND 2.0 BY: wolfpix

A 25-year old man was walking through a field when he was stung by an insect. He developed generalized edema, dyspnea, wheezing and dizziness. He was rushed by a friend to the emergency room, where a diagnosis of anaphylactoid reaction to insect bite was made.

He received a large dose of steroids parenterally and was subsequently advised on a program to taper the steroids over the next one week. Glucocorticoids: Use in Immunosuppression

Heart

James Allen, a 55-year old man with a history of ischemic cardiomyopathy develops increasingly severe congestive heart failure. When he becomes totally incapacitated with a life-expectancy of less than 6 mo., he is placed on the cardiac transplantation list.

Two months later, he receives a heart and is subsequently placed on an immunosuppressive “cocktail” that includes prednisone, 5 mg daily. Glucocorticoids: Use in Androgen Suppression

Hirsutism in a young woman CC:BY-NC-SA BY: C. Matthew Peterson, MD

A 25-year old woman comes in for evaluation of hirsutism present over the past 3 years. The hirsutism is of the androgen type and is associated with acne and irregular menses. Diagnostic studies reveal elevated serum dehydroepiandrosterone (DHEA) and testosterone levels.

She receives Dexamethasone 2.0 mg daily, for seven days and serum DHEA and testosterone levels are measured the 8th day. Adrenocortical Hormones = Steroids

GLUCOCORTICOID MINERALOCORTICOID Cortisol Aldosterone

cortex medulla

Adrenal Effects of Mineralocorticoid on Renal Tubule

Prototypical mineralocorticoid = Aldosterone

Image removed

Aldosterone increases sodium resorption and potassium and hydrogen ion excretion. Prototype of Steroid Compounds

Source: Undetermined Steroids: Structure-function Relationships

A. Hydrocortisone B. Prednisone C. 9--Fluorocortisol

Source: Undetermined

• Double-bond in 1,2 position increases glucocorticoid activity. • Fluoro- group in 9-a position increases mineralocorticoid activity. Steroids

Compound Anti-Inflam. Na-Retain. Duration of Equivalent potency potency action Dose Cortisol 1 1 Short 20 mg

Prednisone 4 0.8 Intermediate 5 mg

9--fluoro- 10 125 Short * cortisone Dexa- 25 0 Long 0.75 mg methasone

Glucocorticoid effects: Dex > Prednisone > Cortisol

Mineralocorticoids: 9--fluorocortisone RULES Glucocorticoid Therapy

Side Effects

Or

“Yes, Virginia, there can be at times ‘Too Much of a Good Thing…’” Glucocorticoid Effects on Calcium & Bone

STEROIDS “BRITTLE BONES”

 Osteoblastic activity  Calcium absorption from gut.  PTH secretion  Osteoclastic activity Steroids “Brittle Bones”

A 60 yo postmenopausal woman was seen in clinic with acute onset of mid-thoracic back pain. She had complained of back pain for the past 2 years and a 2” loss of height. She had been on Prednisone, 10-15 mg daily, for the past 5 years for chronic polymyositis. Radiographic exam of the spine shows compression deformities in several vertebral bodies. CC:BY-NC-ND 2.0 BY: Wellcome Images

Public Domain NIH Chronic Glucocorticoid Therapy & Carbohydrate Metabolism

GLUCOCORTICOIDS

(+)

BLOOD (-) Liver GLUCOSE

(-) MUSCLE

(+) INSULIN

Chronic glucocorticoid therapy may “unmask” diabetes in genetically susceptible individuals. FAT CELL Glucocorticoid Effects on the Central Nervous System

caudate

hippocampus

• Neuronal death or atrophy • Structures affected: Hippocampus, caudate • Neuropsychiatric symptoms: - Cognitive- memory, learning - Mood- irritability, depression - Sleep- insomnia CC:BY-NC-ND BY: Wellcome Images “Steroid Psychosis”

A 42-yo woman with an exacerbation of lupus nephritis was treated with high-dose prednisone for several days. Her nephritis improved markedly; however, she became increasingly euphoric and severely agitated with paranoid ideation and confusion.

Following tapering of the steroid, she returned to her “usual self.” Glucocorticoid Effect on Gastric Function

STOMACH

•  Secretion of HCl and pepsin •  Protective barrier in the gastric mucosa

Glucocorticoid therapy may increase risk of ulcers. Complications of Chronic Exogenous Corticosteroid Use

CRH Hypothalamus (-)

ACTH Pituitary (-)

Cortisol

(+)

Adrenal Gland Complications of Chronic Exogenous Corticosteroid Use

Exogenous CRH Hypothalamus Glucocorticoid (-)

ACTH Pituitary

Cortisol

Exogenous (+) glucocorticoids suppress ACTH- stimulated cortisol secretion. Adrenal Gland Complications of Chronic Exogenous Corticosteroid Use

Exogenous CRH Hypothalamus Glucocorticoid (-)

ACTH Pituitary

ACTH is normally a trophic factor for the adrenals Cortisol

High-dose, long-term glucocorticoid use (+) results in adrenal atrophy from ACTH Adrenal suppression Gland Complications of Chronic Exogenous Corticosteroid Use

Exogenous CRH Hypothalamus Glucocorticoid

ACTH Pituitary

Cortisol

(+)

Adrenal Gland Recovery of Endogenous Cortisol Secretion Following Withdrawal of Exogenous Steroids

Phase 1 Phase 2 Phase 3

HIGH RANGE

NORMAL RANGE ACTH

LOW RANGE

Source: Undetermined months 2 4 6 8 10 12 Full recovery of endogenous cortisol secretion may require up to 18 months following steroid withdrawal. Case #1

A 45-year old woman present with a two-month history of anorexia, nausea, fatigue, dizziness when assuming the upright posture, and increased pigmentation of the skin.

A diagnosis of Addison’s disease (Cortisol and Aldosterone deficiency) is confirmed by appropriate testing.

Treatment is initiated with Cortisol 25 mg. (10/10/5) and 9- fluorocortisol 0.05 mg QD. Corticosteroid Therapy Considerations

• How serious is the underlying disorder? • How long is therapy required? • What is the anticipated effective dose range? • Is patient predisposed to complications? • Which preparation to use? • Alternate day v. every day therapy. • Program for withdrawal. Complications with Prolonged Steroid Therapy

• Retarded longitudinal growth • Steroid myopathy in children* • Hypertension • GI Bleeding • Cataracts • Osteoporosis* • Psychiatric • Diabetes* • Adrenal suppression* • Cushing’s Syndrome

*Complications to remember Things to Remember if Dr. Lash put you to sleep and you’re just waking up…:

Understand:

• Feedback loops regulating cortisol secretion.

• The major physiologic actions of glucocorticoids (cortisol) and mineralocorticoids (aldosterone).

• The major pharmacologic uses of glucocorticoids.

• The major types of glucocorticoids--hydrocortisone, prednisone, dexamethasone, 9-a-fluorocortisol.

• The major side effects of glucocorticoid therapy. Adrenal Steroid Physiology & Pharmacology

Questions? DisordersDisorders ofof thethe AdrenalAdrenal CortexCortex

Logo: All Rights Reserved Regents of the University of Michigan. 2008

Gary D. Hammer, M.D., Ph.D. University of Michigan Ann Arbor, Michigan USA Goals/Objectives

 Remember the basic principles of the HPA axis: homeostatic control of plasma cortisol and aldosterone levels

 Remember the mechanism of action of glucocorticoids and mineralocorticoids

 Understand etiology, clinical features, differential diagnosis, evaluation and therapy of 3 classic adrenal disorders:

Adrenal Insufficiency Cushing’s Syndrome Primary Which Twin is Sick????

Image of patients removed Adrenal Glands in Medical History

Public Domain Public Domain

Andreas Vesalius (1543) Bartholomäus Eustachius (1564) Book Five of De Corporis Humani Fabrica in 1543 glandulae quae renibus incumbent" in 1564 History of Adrenal

 1716: Academie des Sciences of Bordeaux poses the question "Quel est l'usage des glandes surrenales?"

 1845: French thesis on organs of Undetermined function "The adrenal cease(s) to be a secreting gland."

 1855: Thomas Addison monograph “On the constitutional and local effects of disease of the supra-renal capsules,” described 10 cases marked by "anemia . . . feebleness of the heart action . . . a peculiar change of color in the skin occurring in connection with a diseased condition of the ‘suprarenal capsules'.

 In 1945 Nobel Prize Kendall, Pfiffner, and Reichenstein first tested adrenal extracts on a patient with Addison's disease, and the response was prompt and striking. Anatomy of the adrenal glands

Public Domain Wikimedia Commons

Public Domain Seers.gov

Public Domain Wikimedia Commons Histology of the Adrenal Gland

adrenal cortex

CC:BY-NC-SA 1.0 BY: Royal College of Surgeons of Ireland (RCSI)

adrenal medulla

Public Domain Wikimedia Commons Adrenocortical Hormones = Steroids

GLUCOCORTICOID MINERALOCORTICOID Cortisol Aldosterone

cortex medulla

Adrenal Definition of Adrenal Insufficiency

 “inappropriately low” adrenal steroid output

 mineralocorticoids (aldosterone)  glucocorticoids (cortisol)  sex steroids (DHEAS) How Frequent Is Adrenal Insufficiency?

 In general, about 40-60 per million individuals have adrenal insufficiency  30,000-34,000 people in U.S.

Public Domain

CC:BY-NC-SA BY: synnwang Types of Adrenal Insufficiency

CRH Hypothalamus TERTIARY (-)

ACTH Pituitary (-) SECONDARY

Cortisol

(+)

Adrenal Gland PRIMARY Adrenal Insufficiency

1ºadrenal 2ºadrenal 2ºadrenal insufficiency insufficiency insufficiency - hypothalamic CRH + - pituitary ACTH

+ adrenal cortisol adrenal aldosterone adrenal pituitary hypothalami c defect defect defect Adrenal Insufficiency: Age Dependent Prevalence

mean age 40 yo (range 17-72 yo) autoimmune adrenalitis most common in all age groups

children: consider PGA or genetic defect young men: adrenoleukodystrophy

adults and elderly: glucocorticoids for non -adrenal diseases Types of Adrenal Insufficiency

CRH Hypothalamus (-)

ACTH Pituitary (-)

Cortisol

(+)

Adrenal Gland PRIMARY PRIMARY Adrenal Insufficiency

CRH Hypothalamus (-)

ACTH Pituitary Public (-) Domain Cortisol Thomas Addison (1793-1860)

(+) Aldosterone  Autoimmune adrenalitis (PGA I or II) 80% Adrenal Gland  Infections: TB (20% - historically), CMV, fungal  Vascular: hemorrhage, thrombosis, arteritis

 In cancer patients: metastatic cancer to adrenals  In young men: adrenoleukdystrophy

IMPORTANT: In PRIMARY adrenal insufficiency, the adrenals are destroyed, and ALDOSTERONE is affected as well. Adrenal Insufficiency

Autoimmune Adrenal Adrenal Adrenalitis Tuberculosis Hemorrhage

Image of Image of adrenal CC:BY-NC-SA autoimmune tuberculosis BY: University of Alabama at adrenalitis removed Birmingham, Department of Radiology removed

CC:BY-NC-SA BY: University of Alabama at Birmingham, Department of Radiology Metastases in the Adrenal Gland

Skin

Lung Chest

Leukemia Kidney

Ovaries Lymphomas

Public Domain Wikimedia Commons Adrenoleukodystrophy/Adrenomyeloneuropathy

X-LINKED - ONLY IN MALES

PPRESENTATIONRESENTATION -adrenal insufficiency (childhood) -hypergonadotropic hypogonadism (puberty) -spastic paraparesis/demyelination-AMN(20-30 yo) vs cerebral sclerosis-ALD (childhood)

PPATHOPHYSIOLOGY:ATHOPHYSIOLOGY: mutation in Adrenoleukodystrophy protein(ALPD)

ALPD function -pexoxisomal transport protein anchors very long chain AcylCoA synthetase

DDISEASEISEASE - build up of chol. esters w unbranched saturated long chain FAs

TREATMENT: Cortisol replacement Lorenzo’s Oil helps serum level of VLCFA - but no clinical benefit in 3 yr F/U

MUST BE INCLUDED IN w/u of AI in young men and in w/u AI or hypoglycemia in infants Primary Adrenal Insufficiency

Autoimmune adrenalitis results in ADRENAL INSUFFICIENCY

Image of Adrenal Adrenalitis removed

Autoimmune adrenalitis (and therefore its subsequent ADRENAL INSUFFICIENCY) can be found in specific genetic syndromes, POLYGLANDULAR AUTOIMMUNE SYNDROMES Primary Adrenal Insufficiency

PGA I (Polyglandular Autoimmune Syndrome I) autosomal recessive disease- Iranian Jewish heritage starting in childhood

APECED (Autoimmune Polyendocrinopathy-Candidiasis-Ectodermal Dystrophy) autosomal recessive-Finnish heritage starting in childhood

2 of the following occasionally  adrenal insufficiency (<15 yo)  chronic active HepB  hypoparathyroidism (<10yo)  malabsorption  chronic mucocutaneous candidiasis (<5 yo)  cholelithiosis  juvenile onset pernicious  PLUS OFTEN anemia  dental enamel hypoplasia  alopecia/vitiligo  keratopathy/ecdodermal dystrophy  primary hypogonadism  hypothyroidism  diabetes mellitus

AIRE (AutoImmune REgulator) Nat Gen 17: 393398; 399-403 Which Twin is Sick????

Image of patients removed Famous Names in

Addison’s Disease

Public Domain Public Domain Wikimedia Commons

John F. Kennedy Jane Austin (1775-1817) Addison’s Disease & History

Public Domain Wikimedia Commons

1960 Presidential Debate John F. Kennedy vs. Richard M. Nixon Chicago, Ill., September 21, 1960 Adrenal Insufficiency

Autoimmune adrenalitis

 PGA II  usually in middle age females  adrenal insufficiency  hyothyroidism or diabetes mellitus

 *uncertain genetic component  autosomal dominant more likely  HAL-B8 chromosome 6

 PGA III  hypothyroidism  other autoimmune disorder (NOT adrenal insufficiency) Primary Adrenal Insufficiency

SYMPTOMS

Cortisol • Fatigue • Weakness & Malaise CRH Hypothalamus • Anorexia (-) • Nausea and vomiting ACTH Pituitary (-) Aldosterone Cortisol • Dizziness

(+) Aldosterone

Adrenal Gland SIGNS

• Proximal muscle weakness • Orthostatic hypotension • HYPERPIGMENTATION--Primary AI only • HypoNa, HyperK—Primary AI only

aldosterone p450c11B2 - 18 ßHSD

cholesterol 18OHCS sTaR p450c11B2 - 18 OH p450scc p450c11B2 3ßHSD p450c21 pregnenolone progesterone DOCS CS

Hypotension Hyperkalemia Hyponatremia Primary Adrenal Insufficiency (ALDOSTERONE DEFECT ONLY SEEN IN PRIMARY AI not SECONDARY AI)

RENAL TUBULE BLOOD

ALDOSTERONE

H+ Aldosterone increases sodium + K resorption and + Na potassium and K+ hydrogen ion Na+ excretion. So, with aldosterone deficiency: Na+ Cl- GluGlu + - K HCO3 Glucocorticoid Deficiency

cholesterol sTaR p450scc 3ßHSD pregnenolone progesterone

7

7

1

1

c c

0 0 17 OH 17 OH 5 17 OH 5

4

4

p p 3ßHSD p450c21 p450c11B1 17OHPregnenolone 17OHprogesterone DOC cortisol

fatigue

hypotension

hypoglycemia DHEAS Deficiency

cholesterol sTaR p450scc 3ßHSD pregnenolone progesterone 17 OH 17 OH

7 7

1 1

c c

0 0 3ßHSD

5 5

4 4 17OHPregnenolone 17OHprogesterone

p p 17,20lyase 17,20lyase 3ßHSD DHEA androstenedione

sulfatase sulfotransferase

DHEAS Male: fatigue,  mood Female: fatigue,  mood, libidinal dysfunction Adrenal Insufficiency: Hyperpigmentation

Image of Image of patient patient removed hands removed

N Engl J Med Hyperpigmentation of palmar creases 1997;337:1666.

ACTH

Public Domain T. Addison “On the constitutional and MSH local effects of disease of the Source: Undetermined suprarenal capsules” 1855 Types of Adrenal Insufficiency

CRH Hypothalamus (-) ACTH Pituitary SECONDARY (-)

Cortisol

(+)

Adrenal Gland Types of Adrenal Insufficiency

CRH Hypothalamus TERTIARY (-) ACTH Pituitary (-)

Cortisol

(+)

Adrenal Gland Secondary & Tertiary Adrenal Insufficiency

CRH Hypothalamus (-)  Vascular: Postpartum necrosis(Sheehan’s)

ACTH Pituitary  Lymphocytic hypophysitis (-)  Infiltrative diseases: Sarcoidosis, Histiocytosis X Cortisol  Tumor compression  Following surgery or radiation

(+)  Long term glucocorticoid treatment Pharmacologic Dose = more than physiologic replacement Adrenal Gland Secondary Adrenal Insufficiency

CRH Hypothalamus SYMPTOMS (-) SYMPTOMS ACTH Pituitary (-) • Mild malaise, fatigue Cortisol • Proximal muscle weakness

(+) Aldosterone SIGNS

Adrenal Gland • NO hyperpigmentation • NO orthostatic hypotension

SIGNS & SYMPTOMS are generally milder than with primary adrenal insufficiency due to cortisol deficiency ALONE (ie: NO ALDOSTERONE DEFICIENCY) Adrenal Insufficiency

REMEMBER TO DIFFERENCE BETWEEN PRIMARY AI AND SECONDARY AI

PRIMARY ONLY SECONDARY ONLY hyperpigmentation (92-96%) HYPERkalemia (52-64%) associated features (ie can see if PGA) associated features (ie can see if entire pit. involvedinvolved

vitiligo (4%) growth delay hypothyroidism (primary) HA hypogonadism (primary) DI (if stalk involved) hypothyroidism (secondary) hypogonadism (secondary)

*hemorrhage  thromboembolic disease  Coagulopathy  anticoagulant therapy  Waterhouse-Friderichsen Syndrome  Neisseria meningitidis septicemia  Streptococcus pneumoniae,  Pseudomonas aeruginosa  Staphylococcus aureus  Escherichia coli  Haemophilus influenzae  *drugs - increase metabolism GC  phenytoin, phenobarbitol, rifampin  *drugs - decrease production GC  ketoconazole, AG, mitotane, metyrapone

 *withdrawal of exogenous glucocorticoids Adrenal Crisis

 suspect in setting of:  look for:  catecholamine resistant hypotension  hyperpigmentation/decreased pubic hair  hypotension with abd pain  hyperkalemia must r/o adrenal hemorrhage  hyponatremia  hypoglycemia

If the diagnosis is missed, your patient will most likely die Adrenal Insufficiency Diagnostic

SCREENING TEST:

AM CORTISOL: GOAL is to RULE OUT disease

PPrinciplerinciple of test: Cortisol is highest in the AM allowing maximal chance of ruling out disease

-HI AM cortisol RULES OUT DISEASE

-BUT ONLY EXTREMELY LOW AM cortisol is DIAGNOSTIC

MMostost patients are neither EXTREMELY HI or EXTREMELY LOW and require DYMANIC testing Adrenal Insufficiency Diagnostic

DIAGNOSTIC TEST FOR PRIMARY ADRENAL INSUFFICIENCY:

ACTH STIMULATION TEST: GOAL is to RULE IN disease

Principle of test: ACTH stimulates steroidogenesis and secretion of cortisol - normal levels well documented

-Cortisol level after ACTH that is SUBNORMAL is DIAGNOSTIC of AI

-ACTH level that is EXTREMELY HI is CONSISTENT with diagnosis of PRIMARY AI but is NOT DIAGNOSTIC The ACTH Stimulation Test

CRH Hypothalamus CRH Hypothalamus (-)

ACTH Pituitary ACTH (-) Pituitary

No Low or blunted serum Cortisol Cortisol increasecortisol in serum cortisol (+) (+)

Adrenal Adrenal Gland Gland

NORMAL ADRENAL INSUFFICIENCY Synthetic ACTH (Cosyntropin) Adrenal Insufficiency Diagnostic

DIAGNOSTIC TEST FOR SECONDARY ADRENAL INSUFFICIENCY:

INSULIN HYPOGLYCEMIA TEST: GOAL is to RULE IN disease

Principle of test: Insulin results in hypoglycemia that is the strongest stimulus for activation of HPA axis at the level of CRH

Cortisol level after IHT that is SUBNORMAL is DIAGNOSTIC of AI

ACTH level after IHT that is SUBNORMAL is DIAGNOSTIC of SECONDARY AI Diagnosis of Secondary/Tertiary Adrenal Insufficiency

The Insulin Tolerance Test

CRH Hypothalamus INSULIN (-)

ACTH Pituitary (-)

Cortisol Insulin-induced hypoglycemia is a powerful stimulus of the (+) HPA axis

Adrenal Gland Therapy for Adrenal Insufficiency

Therapy

Image of Image of adrenal adrenal gland gland removed removed

Public Domain Public Domain

1543 1564 Guidelines for Management

GUIDING PRINCIPLE: The more severe the stress the more cortisol patient needs

Acute Therapy (significant ill or Adrenal Crisis)

IV fluids Do not wait for labs!!!! IV cortisol: HI DOSE glucose treat underlying precipitating events

Maintenance Therapy Glucocorticoids hydrocortisone ~ 15-25 mg/d titrate to a sense of well being and physical strength avoid weight gain, hypertension, hyperglycemia and osteoporosis Mineralocorticoids fludrocortsone ~0.1 mg/d titrate to salt craving and postural hypotension together with serum K and upper range renin DHEA - Guidelines for Management

GUIDING PRINCIPLE: The more severe the stress the more cortisol patient needs!

Stress Dosing Glucocorticoids

MMinimalinimal no need for supplemental coverage dental work mild or non-febrile illness

Minor 25 mg hydrocortisone - day of procedure (or onset of fever) hernia repair

MModerateoderate 50-75 mg hydrocortisone - day of procedure (or onset of fever) hemicolectomy rapid taper in 1-2 days significant febrile illness

Severe 100-150 mg hydrocortisone - day of procedure (or onset of fever) cardiac surgery rapid taper in 1-2 days

Critically ill 100 mg hydrocortisone i.v. bolus followed by - sepsis 50-100 mg hydrocortisone i.v. q 6-8 hours (or 0.18 mg/kg/hr) 0.05 mg/d fludrocortisone until shock resolves (days to week Discontinuing Glucocorticoids Following Long Term Suppression

GUIDING PRINCIPLE: The more glucocorticoid and the longer treated - the greater chance of long term suppression and atrophy of HPA axis

risk of suppression Low risk: Low dose, short duration or short “bursts” of glucocorticoid High dose and prolonged therapy ( 1-4 weeks) - risk is higher

time course for recovery Larger doses for prolonged periods (months - years) - recovery can take from 9 MONTHS up to 1-2 years

need for taper taper from pharmacologic to physiologic (determined by non-adrenal disease course) taper from physiologic to no treatment (determined by adrenal suppression) DHEA: What is all the fuss?

Marker of aging -??pharmacologic reversal of aging process??

Predictor of morbidity/mortality

Works wonders in rodents -CNS, obesity, diabetes, immunity

Preliminary studies in humans DHEA what is it??

19 carbon (androstane) 5,3-hydroxy,17-keto S04, ester at 3

Source: Undetermined Synthesized by adrenals only in humans and higher primates -obligate precursor of all sex steroids in humans

More synthesized than all other steroids -up to 25 mg/day in adults -major secretion of fetal adrenal

Most secreted as sulfate (DHEA-S) -sulfation is ONLY in ADRENAL (NOT GONAD)

Inactive at androgen receptor DHEA: How Does it Work?

Conversion to androgens -50 mg/d raises testosterone in females

Intrinsic activity of DHEA-S in brain -trophic effects on cultured neurons -GABA, NMDA, sigma receptor-channels

Actions of weird metabolites -concept of NEUROSTEROIDS Case for DHEAS

DHEA + DHEAS major secretory products of adrenal peak in fetal life and adrenarche

Decline throughout adult life to 20-20% by 70-80 yo

Source: TRENDS Advertisement as ANTI-AGING drug In USA : FOOD SUPPLEMENT!!!!!

classic steroid converted to testosterone peripherally neurosteroid directly binding NMDA + GABA receptors Images of DHEA removed Case for DHEAS

Source: TRENDS DHEA in men and women with primary adrenal insufficiency improves mood and well-being, irrespective of the patient's sex.

Source: TRENDS DHEA replacement in women with adrenal insufficiency improved overall well-being and mood, specifically depression, anxiety and both sexual interest and sexual satisfaction. Guidelines for DHEA Treatment in Adrenal insufficiency

Adrenal Androgens only in pts w AI who do NOT feel “normal on replacement GC and MC”

DHEA: 25 mg po q a.m.

-may increase to 50 mg -dictated by response and androgenic side effects -monitor labs DHEAS, androstendione and free test LFTS and lipids at 4 + 12 w Watch Out for Supplements

Steroids are lipophilic Undetermined dosing Undetermined purity

Image of Raw Image of Adrenal Image of Adrenal Adrenal Glandular Plus Glandular Plus Supplements Supplements Supplements removed removed Image of Adrenal Cortex Complex Supplements removed Congenital Adrenal Hyperplasia

Hypothalamus CRH • Genetic block in biosynthetic pathway (-) for cortisol and aldosterone result in primary adrenal insufficiency. ACTH Pituitary (-) • Decreased feedback on hypothalamus Cortisol and pituitary increase CRH and ACTH.

(+) • Increased ACTH further stimulates adrenals and results in shunting and production of precursors. Adrenal Gland • ACTH stimulates growth (HYPERPLASIA) of adrenals. Steroidogenesis

glomerulosa

fasciculata

reticularis

gonad periphery

Public Domain Wikimedia Commons Steroidogenesis

glomerulosa

fasciculata

reticularis

gonad periphery

Public Domain Wikimedia Commons Steroidogenesis

glomerulosa

fasciculata

reticularis

gonad periphery

Public Domain Wikimedia Commons SEVERE P45c21 Deficiency in FEMALE

Image of patient removed

results in androgen excess in utero MILD P45c21 Deficiency in FEMALE

Beard

CC:BY-NC-SA 1.0 BY: C. Matthew Peterson, MD

CC:BY-NC-SA 1.0 BY: C. Matthew Peterson, MD

Cliteromegaly

Hirsuitism

results in androgen excess at puberty Congenital Adrenal Hyperplasia

Important things to remember: • Loss of function of enzyme in steroidogenesis pathway

• “Block” in pathway leads to shunting down alternate paths and abnormal build-up precursors before the block.

• Severe forms lead to virulization of females

• Milder forms (“non-classical”) may lead to hirsuitism and menstrual abnormalities in women.

• Block in pathway may result in adrenal insufficiency during times of stress. Adrenal Excess States Causes of hypercortisolism

 Physiological states  Pathologic states • Pregnancy  Cushing's syndrome  Diabetes mellitus • Stress  Hyperthyroidism • Chronic excessive exercise  Severe chronic disease • Malnutrition  Glucocorticoid resistance  Psychological states  Anorexia nervosa  Panic disorder  Melancholic depression  Obsessive-compulsive disorder

 PHARMACOLOGIC USE OF GLUCOCORTICOIDS Cushing’s Syndrome

Public Domain Public Domain Harvey Cushing (far left) in 1895 during his DR. HARVEY WILLIAMS CUSHING House Pupilship (internship) at (1869-1939) Massachusetts General Hospital.

Cushing HW. The basophil adenomas of the pituitary body and their clinical manifestations (pituitary basophilism). Bulletin of the Johns Hopkins Hospital. 1932;50:137-95

His research on the pituitary body gained him an international reputation, and he was the first to ascribe to pituitary malfunction a type of obesiobesityty of the face and trunk now known as Cushing's disease, or Cushing's syndrome. Cushing‘s Syndrome

 All types of Cushing’s Syndrome HI CORTISOL (urine and serum) Absent circadian rhythm

 Adrenal Cushing’s syndrome is autonomous and therefore has LOW ACTH  Only ACTH-dependent Cushing’s (by definition) has HI ACTH Cushing’s Syndrome

ACTH independent Cushings ACTH dependent Cushing s pituitary ectopic ectopic adrenal ACTH ACTH CRH Cushing s Cushing s Cushing s Cushing s

- hypothalamic CRH ectopic + CRH - pituitary ACTH ectopic ACTH + adrenal cortisol adrenal pituitary ectopic defect defect defect Cushing’s Syndrome

Exogenous GC administration Pituitary adenoma

ACTH-dependent Ectopic ACTH Endogenous (CRH) syndrome hypercortisolism

Adrenal ACTH- adenoma independent Adrenal carcinoma Cushing’s Syndrome

 ACTH-dependent Cushing’s Syndrome pituitary adenoma-ACTH (60%) Image of pituitary Ectopic hormone (10%) glands removed ACTH CRH

all result in bilateral adrenal hyperplasia Types of Cushing’s Syndrome

PITUITARY Hypothalamus ADENOMA CRH (-) Cushing’s Disease ACTH Pituitary (“pituitary Cushings”): (-) hypercortisolism from a pituitary adenoma Cortisol HARVEY

(+)

Adrenal Gland

Public Domain Normal Pituitary

GNU Free documentation license version 1.2 Wikimedia Commons

Source: Undetermined Pituitary Cushing‘s DISEASE

Source: Undetermined normal Source: Undetermined Cushing’s disease Ectopic Cushing’s

Hypercortisolism from CRH Hypothalamus Ectopic production of (-) ACTH or CRH by tumor. ACTH Pituitary (-) Cortisol Images of oat cell in lung removed ACTH Causes • Bronchial carcinoid • Oat cell carcinoma • Thymic carcinoid • Pheochromocytoma Small (Oat) cell ca of lung Adrenals • Medullary thyroid ca Cushing’s Syndrome

 ACTH-independent Cushing’s Syndrome  adrenal cortical neoplasm  adenoma  carcinoma  primary adrenal hyperplasia

CC:BY-NC-SA 1.0 BY: Cornell University Adrenal Cushing’s

Adrenal Causes: CRH Hypothalamus Hypercortisolism from a (-) adrenal adenoma or carcinoma ACTH Pituitary (-) Cortisol Because ACTH is suppressed, the rest of the adrenal and contralateral (+) gland are atrophied.

Adrenal Contralateral Adrenal Adrenal adenoma or carcinoma Cushing’s Syndrome

CLINICAL MANIFESTATIONS of CORTISOL EXCESS

 increased protein catabolism = striae, bruising, delayed wound healing, muscle wasting

 increased glucose production = DM

 redistribution of fat = truncal obesity ACTH dependent ONLY  Pigmentation (MSH)  bone breakdown = osteoporosis ACTH dependent or Mixed Adrenal  facilitation of catechol synthesis = hypertension  Androgen excess  Terminal hair hirsuitism  anti-inflammatory = opportunistic infections  Acne  Inhibition of HPG axis = amenorrhea, impotence  Irregular menses  balding  CNS effects(limbic/hippocampus) = depression and memory difficulties Cushing‘s Syndrome

 Physical examination:  adiposity  moon face, plethora  (pseudo-) gynecomastia  striae

Image of patient Image of patient with moon face / with striae on arms plethora removed removed Cushing‘s Syndrome

 Acanthosis nigricans  Purple striae

Image of patient Image of patient Image of patient with acanthosis with striae on with striae on nigricans on armpit abdomen removed abdomen removed removed Cushing‘s Syndrome

 Myopathy  Proximal muscle wasting Images of myopathic  Osteoporosis patient removed  Oligo-Amenorrhea/Impotence  Psychiatric Symptoms  depression, mania (Steroid psychoses) ACTH-Dependent Pituitary Cushing‘s Disease

 Symptoms due to pituitary mass  bitemporal hemianopsia  pituitary insufficiency  HA Cushing‘s Syndrome: Diagnosis

 Diagnosis  First diagnose CORTISOL EXCESS elevated 24 hr urine cortisol < 100 mg/24 hr

 Then diagnose PATHOLOGIC CORTISOL EXCESS r/o physiologic causes which suppress normally with low-dose DEX (Cort < 5 mg/dl)

 ACTH dependent or NOT Measure ACTH level if DETECTABLE > 9 pg/ml - must be ACTH dependent (if NOT DETECTABLE < 9 pg/ml - must be ACTH independent) Cushing’s Syndrome: Low-dose DEX suppression

Low- Dose Dose CRH Hypothalamus DEX (-)

ACTH Pituitary ACTH (-)

Cortisol Ectopic tumor

(+) ACTH

Adrenal Gland

Low-dose Dex will suppress ACTH secretion in: -normal patients -physiologic hypercortisolism (stress)

Low-dose Dex will NOT suppress ACTH secretion in: -ACTH dependent Cushing’s syndrome (pituitary adenoma or ectopic ACTH producing tumors) -ACTH independent Cushing’s syndrome (adrenal tumors) Cushing‘s Syndrome: Diagnosis

 Diagnosis  First diagnose CORTISOL EXCESS elevated 24 hr urine cortisol < 100 mg/24 hr

 Then diagnose PATHOLOGIC CORTISOL EXCESS r/o physiologic causes which suppress normally with low-dose DEX (Cort < 5 mg/dl)

 ACTH dependent or NOT Measure ACTH level if DETECTABLE > 9 pg/ml - must be ACTH dependent (if NOT DETECTABLE < 9 pg/ml - must be ACTH independent) ACTH-DEPENDENT Cushing’s Syndrome

Is it pituitary or ectopic????

 High dose DEX SUPPRESSION TEST  Pituitary Cushing’s may suppress to high dose DEX  Ectopic NEVER suppresses to high dose DEX

 Inferior Petrosal sinus Sampling  Pituitary Cushing’s - find HI ACTH near pituitary and low in the periphery  Ectopic Cushing’s - find HI ACTH in the periphery and low near pituitary

 IMAGE the pituitary Cushing’s Syndrome: Hig-dose DEX suppression

HIGH- Dose Dose CRH Hypothalamus DEX (-)

Most pituitary adenomas that ACTH Pituitary secrete ACTH can still be (-) inhibited by REALLY REALLY ACTH HIGH glucocorticoids (ie more that produced their Cortisol diseased HPA axis)

Therefore, HIGH-dose dexamethasone will NOT (+) suppress ACTH from ectopic tumors. Adrenal Gland

High-dose Dex will suppress ACTH secretion in: -ACTH dependent Cushing’s syndrome (pituitary adenoma)

High-dose Dex will NOT suppress ACTH secretion in: -ACTH dependent Cushing’s syndrome (ectopic tumors) Cushing’s Syndrome: Diagnosis

HIGH- Dose Dose CRH Hypothalamus DEX (-)

Pituitary Most ectopic ACTH- ACTH producing tumors secrete (-) ACTH independently from regulation by glucocorticoids. Cortisol Cortisol Ectopic Therefore, HIGH-dose tumor dexamethasone will NOT suppress ACTH from ectopic tumors. (+) ACTH

Adrenal Gland

High-dose Dex will suppress ACTH secretion in: -ACTH dependent Cushing’s syndrome (pituitary adenoma)

High-dose Dex will NOT suppress ACTH secretion in: -ACTH dependent Cushing’s syndrome (ectopic tumors) Imaging in Cushing Syndrome

 ADRENAL CT findings  Pit MRI findings  adrenals small = ?  Mass or no mass  one adrenal large and 1 small =? (some pituitary corticotrope  Both adrenals large=? tumors are too small to be seen on MRI)

 Search for ectopic ACTH or CRH producing tumor  Lung: Bronchial Carcinoid and SCC 50%  Thymic Carcinoid (epithelial thymoma 10%  Pancreatic Islet Cell Tumor 10%  Pleochromocytoma 10%  Abdominal Carcinoids 5%  Medullary Thyroid Carcinoma 5% Cushing’s Syndrome Treatment

 adrenal adenoma  pituitary adenoma  resection transphenoidal resection (TSR)  cortisol replacement cortisol replacement  if not curative if not curative  XRT XRT  bilateral adrenalectomy bilateral adrenalectomy  adrenolytic therapy adrenolytic therapy  mitotane mitotane  ketoconazole mitotane ketoconazole

 Ectopic ACTH or CRH Find the tumor!!!!!!!!!!! if not curative bilateral adrenalectomy adrenolytic therapy mitotane ketoconazole Cushing‘s Syndrome

Image of patient before treatment with Cushing’s Syndrome removed

Image of patient with Cushing’s after treatment Syndrome removed Cushing‘s Syndrome

Public Domain Wikimedia Commons Public Domain Wikimedia Commons CC:BY 2.0 BY: tajai

dogs Ferrets horses UM Endocrinology in Adrenal History: Conn Syndrome

Image of Jerome Image of patient Conn removed removed

CC:BY 2.0 BY: Michael Feldman, MD Jerome Conn, M.D.

Conn JW. , a new clinical syndrome. J Lab Clin Med. 1955;45:3-17 Primary Aldosteronism

 Clinical Presentation Manifestations of HYPOKALEMIA and HTN LOW K neuromuscular paresthesias weakness tetany Renal Polyuria Carbohydrate abnormal GTT HTN usually not malignant early in disease may have HTN with NORMAL K Causes of Hyperaldosteronism

Definition: syndrome of inappropriate excessive secretion of aldosterone by adrenal gland kidney (JGA) renin IVV + Na + IVV + + K angiotensinogen angiotensin a ngiotensin II + AII-R ----> aldosterone + MR H liver adrenal + (glomerulosa) K (serum) ACE met alk lung kidney (distal tubule)

Source: Undetermined

An increase in aldosterone ACTION can theoretically result from ANY defect in RAA pathway

-LOW IVV (real or perceived by kidney in renal artery stenosis) -JGA renin tumor -ACE polymorphisms -overproduction of AII by renal tumors -ADRENAL overproduction of ALDO -constitutive MR or Na channel Primary Aldosteronism

primary hyperaldosteronism (HI ALDO/LOW RENIN)  ZG Aldo tumor 70%  ZG Aldo hyperplasia 30%

 rare/rare/rare Congenital adrenal hyperplasia <1% (p450c11ß, p450c17) ACE polymorphisms < 1% AII overproduction < 1%

secondary hyperaldosteronism (HI ALDO/HI RENIN)  JGA renin tumor <1%  renal artery stenosis <1%

apparent mineralocorticoid excess (LOW ALDO/LOW RENIN) (downstream of ALDO)  constitutively active MR <1%  Na/K/H channel <1%  licorice <1% Primary Aldosteronism

Consider in patients with:

-New HTN -HTN with LOW K

EVEN THOUGH it only accounts for 0.5% of all HTN

BECAUSE-BECAUSE- IFIF YOUYOU NEVERNEVER THINKTHINK OFOF THIS--THIS-- ---YOU---YOU WILLWILL NEVERNEVER FINDFIND IT!!!IT!!! Primary Aldosteronism

 Work-Up  R/O other causes of LOW K LOW intake (diet) HI output N/V/D Diuretic use with loops + thiazides

 24 h Urine ALDO If LOW- pt does not have PRIMARY ALDO IF HI (>10 ug/day) check RENIN level (suppressed < 1 ng/ml/hr) If RENIN HI ----JGA renin tumor or RAS If RENIN LOW---- PRIMARY HYPERALDO

 IF NECESSARY (ie AMBIGUOUS) Volume expand to see if can suppress RAA  If can suppress --essential HTN Adrenal Zona Glomerulosa Adenoma

Image of Adrenal gland removed Primary Aldosteronism

 IMAGING and TREATMENT CT scan Adenoma unilateral ADX NO adenoma selective venous cath to measure ALDO rt vs lt If unilateral elevation-small adenoma If no lateralization-bilateral hyperplasia Medical trt with spironolactone or amiloride bilateral ADX Adrenocortical Carcinoma

 Larger adrenal mass

 High probability NOT benign if >5 cm in diameter

 Image of Adrenal  Development of Cushingoid gland removed features usually very rapid (several months rather than years)

 Often associated with elevated DHEA-sulfate and virulization Remember:

EndocrineEndocrine disordersdisorders areare NOTNOT diagnoseddiagnosed byby meansmeans ofof imaginingimagining studies.studies. BiochemicalBiochemical conconfifirmationrmation mustmust comecome fifirstrst beforebefore imaginingimagining isis performed.performed. Public Domain Wikimedia Commons “Even our destiny is determined by our endocrine glands.”

Albert Einstein